Institute of Human Genetics, University Hospital Erlangen-Nuremberg, Erlangen, Germany.
Hum Mol Genet. 2012 Mar 15;21(6):1336-49. doi: 10.1093/hmg/ddr572. Epub 2011 Dec 8.
The molecular events responsible for obstruction of aqueous humor outflow and the loss of retinal ganglion cells in glaucoma, one of the main causes of blindness worldwide, remain poorly understood. We identified a synonymous variant, c.765C>T (Thr255Thr), in ankyrin repeats and suppressor of cytokine signaling box-containing protein 10 (ASB10) in a large family with primary open angle glaucoma (POAG) mapping to the GLC1F locus. This variant affects an exon splice enhancer site and alters mRNA splicing in lymphoblasts of affected family members. Systematic sequence analysis in two POAG patient groups (195 US and 977 German) and their respective controls (85 and 376) lead to the identification of 26 amino acid changes in 70 patients (70 of 1172; 6.0%) compared with 9 in 13 controls (13 of 461; 2.8%; P = 0.008). Molecular modeling suggests that these missense variants change ASB10 net charge or destabilize ankyrin repeats. ASB10 mRNA and protein were found to be strongly expressed in trabecular meshwork, retinal ganglion cells and ciliary body. Silencing of ASB10 transcripts in perfused anterior segment organ culture reduced outflow facility by ∼50% compared with control-infected anterior segments (P = 0.02). In conclusion, genetic and molecular analyses provide evidence for ASB10 as a glaucoma-causing gene.
导致青光眼(一种全球主要致盲原因之一)房水流出受阻和视网膜神经节细胞丧失的分子事件仍未被充分理解。我们在一个具有原发性开角型青光眼(POAG)遗传特征并定位于 GLC1F 基因座的大型家族中发现了锚蛋白重复和细胞因子信号抑制因子盒蛋白 10(ASB10)中的同义变异 c.765C>T(苏氨酸 255 变为苏氨酸)。该变异影响外显子剪接增强子位点,并改变了受影响家族成员的淋巴母细胞中的 mRNA 剪接。在两个 POAG 患者组(195 名美国人和 977 名德国人)及其各自的对照组(85 名和 376 名)中进行系统的序列分析,导致在 70 名患者(1172 名中的 70 名;6.0%)中鉴定出 26 个氨基酸变化,而在 13 名对照者(461 名中的 9 名;2.8%;P=0.008)中发现 9 个氨基酸变化。分子建模表明,这些错义变异改变了 ASB10 的净电荷或使锚蛋白重复不稳定。ASB10mRNA 和蛋白在小梁网、视网膜神经节细胞和睫状体中强烈表达。与对照感染的前节相比,在前节灌流器官培养物中沉默 ASB10 转录本可使房水流出率降低约 50%(P=0.02)。总之,遗传和分子分析为 ASB10 作为致青光眼基因提供了证据。